Vesicular Pemphigoid – Ultrastructural and Immunoelectron Microscopic Study

Hayakawa, Kazuhito; Shimizu, Hiroshi; Amagai, Masayuki; Harada, Takashi; Nishikawa, Takeji; Yamasaki, Yuichiro

doi:10.1159/000248430

Cited by 5 publications

(1 citation statement)

References 10 publications

(12 reference statements)

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“…VP has been reported by several authors 3–6 . Apart from the size of lesions, clinical and laboratory findings are similar to those in patients with BP.…”

supporting

confidence: 78%

The application of topical tacrolimus in vesicular pemphigoid

Chuh¹

2004

Br J Dermatol

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4 Lapins J, Emtestam L, Marcusson JA. Angiokeratomas in Fabry's disease and Fordyce's disease: successful treatment with copper vapour laser. Acta Derm Venereol (Stockh) 1993; 73: 133-5. 5 Möhrenschlager M, Braun-Falco M, Ring J, Abeck D. Fabry disease: recognition and management of cutaneous manifestations. Am J Clin Dermatol 2003; 4: 189-96.The application of topical tacrolimus in vesicular pemphigoid SIR, Bullous pemphigoid (BP) occurs mainly in elderly patients, and is associated with high morbidity and sometimes mortality. 1 Advanced patient age and high doses of systemic corticosteroids are major risk factors for mortality. 2 Vesicular pemphigoid (VP) is an uncommon variant of BP.We describe a patient with VP for whom topical steroids did not achieve good symptomatic control, but the use of topical tacrolimus has spared the need for long-term systemic treatment.A 69-year-old retired businessman had generalized pruritus for 6 months. His past health was good. He was a nonsmoker and social drinker. He had been taking hypnotic agents occasionally as prescribed by a psychiatrist. His medication history was otherwise unremarkable. Examination revealed a lean elderly man with no pallor and no jaundice. Generalized excoriation marks were evident. A tense and firm-topped vesicle 4 mm in diameter was seen on the left side of his chest. Dermatoscopic examination revealed clear fluid in the vesicle, with inflammatory vascular changes evident at the periphery of the lesion. Nikolsky's sign was negative. The patient stated that similar vesicles had erupted before, and pointed out various healing or healed lesions with postinflammatory hyperpigmentation on the chest wall, axillae, abdomen, forearms, thighs and legs. The lesions were not arranged in groups. Palmoplantar surfaces and mucous membranes were uninvolved.Full blood count, fasting glucose, liver, renal and thyroid function tests were normal. Antinuclear antibodies and rheumatoid factor were negative. Biopsy of the active lesion revealed a subepidermal vesicle. Exocytosis of inflammatory cells with a predominance of eosinophils was seen. Mild epidermal spongiosis and focal parakeratosis were noted. Direct immunofluorescence for IgG and C3 revealed positive staining along the basement membrane. Staining for IgA and IgM was negative. Staining for fibrin was faintly positive along the basement membrane and base of the vesicle.The clinical, histopathological and immunofluorescence findings were all consistent with VP. More fresh intact vesicles were noted at subsequent visits. We prescribed oral prednisolone 20 mg daily (0AE4 mg kg )1 daily) with oral cimetidine 400 mg daily. The systemic corticosteroid was tapered to a dose of 5 mg on alternate days (0AE05 mg kg )1 daily). Topical clobetasol propionate 0AE05% ointment was then commenced. However, new lesions continued to erupt. We then commenced topical tacrolimus ointment twice daily, first at 0AE03% and then at 0AE1%. After 2 weeks, the oral prednisolone was stopped altogether. No further new lesions were seen. Th...

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“…VP has been reported by several authors 3–6 . Apart from the size of lesions, clinical and laboratory findings are similar to those in patients with BP.…”

supporting

confidence: 78%

The application of topical tacrolimus in vesicular pemphigoid

Chuh¹

2004

Br J Dermatol

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Autoimmune Subepidermal Bullous Diseases of the Skin and Mucosae: Clinical Features, Diagnosis, and Management

Amber

Murrell

Schmidt

et al. 2017

Clinic Rev Allerg Immunol

175

191

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Autoimmune subepidermal blistering diseases of the skin and mucosae constitute a large group of sometimes devastating diseases, encompassing bullous pemphigoid, gestational pemphigoid, mucous membrane pemphigoid, epidermolysis bullosa acquisita, and anti-p200 pemphigoid. Their clinical presentation is polymorphic. These autoimmune blistering diseases are associated with autoantibodies that target distinct components of the basement membrane zone of stratified epithelia. These autoantigens represent structural proteins important for maintenance of dermo-epidermal integrity. Bullous pemphigoid (BP) is the most common subepidermal autoimmune blistering disease of the skin and mucosae. Although the disease typically presents with a generalized blistering eruption associated with itch, atypical variants with either localized bullous lesions or "non-bullous" presentations are observed in approximately 20% of patients. A peculiar form of BP typically associated with pregnancy is pemphigoid gestationis. In anti-p200 pemphigoid, patients present with tense blisters on erythematosus or normal skin resembling BP, with a predilection for acral surfaces. These patients have antibodies targeting the 200-kDa basement membrane protein. Epidermolysis bullosa is a rare autoimmune blistering disease associated with autoantibodies against type VII collagen that can have several phenotypes including a classical form mimicking dystrophic epidermolysis bullosa, an inflammatory presentation mimicking BP, or mucous membrane pemphigoid-like lesions. Mucous membrane pemphigoid (MMP) is the term agreed upon by international consensus for an autoimmune blistering disorder, which affects one or more mucous membrane and may involve the skin. The condition involves a number of different autoantigens in the basement membrane zone. It may result in severe complications from scarring, such as blindness and strictures. Diagnosis of these diseases relies on direct immunofluorescence microscopy studies and immunoserological assays. Management of affected patients is often challenging. We will here review the clinical and immunopathological features as well as the pathophysiology of this group of organ-specific autoimmune diseases. Finally, we will discuss the diagnostic approach and the principles of management in clinical practice.

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Overlapping cutaneous disorders related to dermatitis herpetiformis

Kaplan

Callen

1991

Clinics in Dermatology

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Vesicular Pemphigoid – Ultrastructural and Immunoelectron Microscopic Study

Cited by 5 publications

References 10 publications

The application of topical tacrolimus in vesicular pemphigoid

The application of topical tacrolimus in vesicular pemphigoid

Autoimmune Subepidermal Bullous Diseases of the Skin and Mucosae: Clinical Features, Diagnosis, and Management

Overlapping cutaneous disorders related to dermatitis herpetiformis

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